| Summary: | Background: Trigeminal trophic syndrome is a rare complication of peripheral or central damage to the trigeminal nerve characterized by anesthesia, paresthesia and a secondary persistent facial ulceration. Methods: We describe the case of a 40-year-old woman with previous history of Le Fort I osteotomy for a class III malocclusion who developed trigeminal trophic syndrome. Atypically, the cutaneous symptoms appeared bilaterally and 8 years after surgery. Results: Differential diagnosis was based on clinical history, tissue biopsy and serologic evaluation. Atypical findings could be linked to the surgical burdens of Le Fort I osteotomy, a procedure characterized by a bilateral incision on the maxillofacial bones with a reasonable probability of causing a bilateral injury of the peripheral branches of the trigeminal nerve. Conclusion: Although the long delay between trigeminal trophic syndrome onset and surgery and the absence of adequate medical evidence cannot confirm a link with previous surgery in this case, the increasing number of maxillofacial surgery cases suggests that this complication may be more frequent in the next decades, and thus, involved specialists should be aware of this condition as a possible complication of maxillofacial surgery procedures.
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